Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts Login 
Home Print this page Email this page Small font size Default font size Increase font size Users Online: 173


 
 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 9  |  Issue : 1  |  Page : 26-31

Pancreatico-pleural Fistula: Case Series


Institute of Medical Gastroenterology, Madras Medical College, Rajiv Gandhi Government General Hospital, Chennai, Tamil Nadu, India

Date of Web Publication12-Mar-2018

Correspondence Address:
Manoj Munirathinam
No. 14, Nolambur Phase 2, Mogappair West, Chennai - 600 037, Tamil Nadu
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jde.JDE_23_17

Rights and Permissions
  Abstract 


Pancreatico-pleural fistula is a rare but serious complication of acute and chronic pancreatitis. The pleural effusion caused by pancreatico-pleural fistula is usually massive and recurrent. It is predominately left-sided but right-sided and bilateral effusion does occur. We report four cases of pancreatico-pleural fistula admitted to our hospital. Their clinical presentation and management aspects are discussed. Two patients were managed by pancreatic endotherapy and two patients were managed conservatively. All four patients improved symptomatically and were discharged and are on regular follow-up. Most of these patients would be evaluated for their breathlessness and pleural effusion delaying the diagnosis of pancreatic pathology and management. Hence, earlier recognition and prompt treatment would help the patients to recover from their illnesses. Pancreatic pleural fistula diagnosis requires a high index of suspicion in patients presenting with chest symptoms or pleural effusion. Extremely high pleural fluid amylase levels are usual but not universally present. A chest X-ray, pleural fluid analysis, and abdominal imaging (magnetic resonance cholangiopancreatography/magnetic resonance imaging abdomen more useful than contrast-enhanced computed tomography abdomen) would clinch the diagnosis. Endoscopic retrograde cholangiopancreatography with stent or sphincterotomy should be considered when pancreatic duct (PD) reveals a stricture or when medical management fails in patients with dilated or irregular PD. Surgical intervention may be indicated in patients with complete disruption of PD or multiple strictures.

Keywords: Endoscopic retrograde cholangiopancreatography, pancreatic pseudocyst, pancreatico-pleural fistula, pancreatitis, pleural effusion


How to cite this article:
Munirathinam M, Thangavelu P, Kini R. Pancreatico-pleural Fistula: Case Series. J Dig Endosc 2018;9:26-31

How to cite this URL:
Munirathinam M, Thangavelu P, Kini R. Pancreatico-pleural Fistula: Case Series. J Dig Endosc [serial online] 2018 [cited 2019 Aug 21];9:26-31. Available from: http://www.jdeonline.in/text.asp?2018/9/1/26/227140




  Introduction Top


Pancreatico-pleural fistula (PPF) is a rare complication of pancreatitis secondary to posterior disruption of the pancreatic duct (PD). The fistulous tract ascends into the pleural cavity and gives rise to large volumes of pleural fluid. Pleural effusion caused by pancreatico-pleural fistula tends to be massive and recurrent. They are predominately left-sided but right-sided and bilateral effusion can also occur. Here, we discuss the four cases of pancreatico-pleural fistula who were admitted to our hospital.


  Case Reports Top


Case 1

A 45-year-old male, a chronic alcoholic was referred to our institute with the complaints of abdominal pain and breathlessness for the past 20 days. He also had cough and copious watery expectoration which was at times blood stained. On physical examination, he was very tachypneic (respiratory rate was 26/min) and breath sounds were diminished on the right hemithorax. He also had minimal free fluid in the abdomen. There was no organomegaly. The patient was apparently normal 6 months back when he was admitted to a local hospital for ethanol-related acute pancreatitis. He was administered analgesics and intravenous fluids and was managed conservatively. After 3 weeks, he was rehospitalized for severe abdominal pain. Magnetic resonance imaging (MRI) abdomen showed resolving acute necrotizing pancreatitis with hemorrhagic pseudocyst and pseudoaneurysm with surrounding inflammatory exudates. He was pale with hemoglobin of 4 g% and was transfused with packed red cells. Computed tomography (CT) angiography showed evidence of two irregular pseudoaneurysms, one near the pancreatic neck measuring 2.3 cm × 1.2 cm arising from the gastroduodenal artery and another inferior to the pancreatic bed measuring 1.8 cm × 1.4 cm arising from the splenic artery. A large hematoma was seen in the peripancreatic region extending into the lesser sac, and another large hematoma was seen tracking into the right intrahepatic region. Coil embolization of the aneurysm arising from the gastroduodenal artery and glue embolization of the other aneurysm was done. The patient responded well and was discharged.

A couple of weeks later, he developed abdominal pain and severe breathlessness. He had cough and copious watery expectoration which was at times blood stained. Of the imaging contrast-enhanced computed tomography (CECT), chest revealed massive right-sided pleural effusion and CECT abdomen showed walled off necrosis and multiple fluid collections in the abdomen. A pigtail catheter was placed ultrasonographically, and the abdominal collection was drained. Meanwhile, an intercostal drain was placed in the right-side chest and pleural fluid was removed. He improved symptomatically and was discharged but again developed the same symptoms and was referred to our institution.

We did a complete blood analysis. His serum amylase and lipase were elevated (544 IU/L and 665 IU/L, respectively). The total count was marginally elevated (13,800/cumm). Renal function tests and liver function tests were within normal limits. The pleural fluid analysis showed a high protein (4.8 g/dl) and pleural fluid amylase level of 9865 IU/L. CECT chest [Figure 1]a and abdomen showed massive right-sided pleural effusion with passive collapse of the underlying lung parenchyma, diffuse edema and enlargement of the pancreatic parenchyma with necrosis in the neck region, and pseudocyst formation involving lesser sac and communicating into the posterior mediastinum with rupture into the pleural cavity with pancreatico-pleural fistula. Magnetic resonance cholangiopancreatography (MRCP) also demonstrated the ductal leak [Figure 1]b. The sputum analysis showed very high values of amylase and lipase and plenty of red blood cells confirming the fistulous communication between the pancreas and the right pleura/bronchial tree.
Figure 1: (a) Computed tomography chest showing massive right-sided pleural effusion. (b) Magnetic resonance cholangiopancreatography picture demonstrating the pancreatic ductal leak and dilated pancreatic duct. (c) Endoscopic retrograde cholangiopancreatography picture demonstrating the contrast entering the right bronchial tree on further injection of the contrast. (d) Computed tomography image showing almost complete resolution of the right pleural effusion at the time of discharge

Click here to view


Initially, the patient had nasojejunal tube intubation and was instituted enteral nutrition. He was started on antibiotics and injection octreotide. After initial stabilization, the patient was submitted for endoscopic retrograde cholangiopancreatography (ERCP). PD cannulation was successful and pancreatogram revealed ductal leak at the level of gene. The fistulous tract could be demonstrated in the ERCP [Figure 1]c and was tracking upward to the right pleural space. On further contrast injection, the bronchial tree could be visualized, and the patient had cough probably due to the irritation of the respiratory mucosa. An attempt was made to place the guide wire into the PD across the leak but it was repeatedly entering into the fistulous tract. Hence, pancreatic sphincterotomy alone was done and the pancreatic juice was found draining freely.

Following the procedure, the patient gradually improved. His breathlessness was settled. The expectoration changed from bloody copious amounts to mucoid smaller amounts. The breath sounds in the right hemithorax became audible and was bronchial in quality suggesting associated consolidation. The abdominal pain was reduced. The patient's general condition was improved. He was initially started on liquid feeds and later tolerated solid diet too. A repeat CT [Figure 1]d revealed resolution of the right-sided effusion with the right lower lobe consolidation and resolution of the collections in the abdomen.

Case 2

A 70-year-old male was admitted to our hospital with abdominal pain radiating to back and breathlessness following a blunt injury to the abdomen. He was not an alcoholic or smoker. On examination, he had minimal free fluid in the abdomen with epigastric tenderness. The breath sounds were absent in the lower left hemithorax. A chest X-ray showed moderate left-sided pleural effusion. The serum amylase and lipase were elevated. The pleural fluid analysis revealed elevated amylase and lipase levels. An MRI abdomen/MRCP revealed well-defined fluid collection measuring 10 cm × 3.2 cm × 2.8 cm extending from the region of the tail of pancreas communicating to the left pleural space through a defect in the dome of diaphragm measuring 1.5 cm implicating a PPF [Figure 2]. The patient was started on injection octreotide, and an intercostal drainage (ICD) tube was placed in the left chest for drainage. An ERCP was attempted but there was a large periampullary diverticulum PD cannulation was not successful. However, the patient improved with conservative management. The ICD secretions gradually decreased and it was removed after 10 days. The patient was discharged and is on regular follow-up.
Figure 2: Magnetic resonance cholangiopancreatography demonstrating fluid collection measuring 10 cm × 3.2 cm × 2.8 cm extending from the region of the tail of pancreas communicating to the left pleural space

Click here to view


Case 3

A 28-year-old male was referred to our institute with complaints of severe abdominal pain for the past 2 weeks. The pain was pulling type in the epigastric region radiating to back, increased on food intake, and relieved on stooping forward. There was no abdominal distension. There was no swelling of legs or decreased urine output. He complained of breathlessness on exertion. There was no steatorrehea. There was loss of weight and loss of appetite. There was minimal cough with expectoration. He had similar episodes of abdominal pain in the past, for which he was treated conservatively. He was admitted for acute pancreatitis in a local hospital 3 weeks back and was managed conservatively and discharged. He was a smoker and an alcoholic for the past 5 years. On examination, the patient was tachypneic with a respiratory rate of 24/min, pale. Abdomen examination revealed epigastric tenderness without any organomegaly of significant free fluid in the abdomen. There were absent breath sounds in the right hemithorax and the trachea was shifted to the left. Blood investigations revealed a normal renal function tests and liver function tests. The serum amylase and lipase were elevated. A chest X-ray revealed massive right-sided pleural effusion. A CECT abdomen showed small areas of necrosis in the head of pancreas with body and tail being atrophic. Small peripancreatic fluid collection was seen. The fluid collection seen was extending to the posterior mediastinum through the esophageal hiatus posterior to inferior vena cava and the collection appeared to be continuous with the right pleural space [Figure 3]a. Massive right-sided pleural effusion with collapse of the right lung was seen. These features were suggestive of chronic pancreatitis with posterior mediastinal pseudocyst and right-sided massive pleural effusion with a high possibility of pancreatico-pleural fistula. An ICD drain was placed in the right chest. An MRCP further confirmed the diagnosis of a PPF [Figure 3]b. The patient was then subjected to ERCP. Pancreatogram demonstrated the leak in the body of the pancreas. PD was stented using 7Fr 8 cm single pigtail stent [Figure 3]c. Free flow of pancreatic juice was noted. The patient improved significantly. The ICD was removed 8th-day post ERCP. The patient is on regular follow-up with us.
Figure 3: (a) Computed tomography picture showing massive right pleural effusion with mediastinal pseudocyst and communication into right pleural cavity. (b) Magnetic resonance cholangiopancreatography demonstrating the ductal leak and the presence of pseudocyst. (c). Endoscopic retrograde cholangiopancreatography picture demonstrating the successful pancreatic duct cannulation and guide wire placement bridging the leak

Click here to view


Case 4

A 37-year-old male who was a chronic alcoholic and smoker presented to us with breathlessness for 4 weeks. On examination, he had decreased breath sounds with crepitations in the right hemithorax. CT chest showed moderate right-sided pleural effusion. Pleural fluid protein was high (4.3 g/dl) suggesting an exudative effusion. The amylase and lipase levels were grossly elevated. An MRCP revealed pancreatic parenchymal atrophy in the distal body and tail of pancreas with prominent main PD (MPD) and side ducts. Well-defined pancreatic fluid collection 3.8 cm × 2.1 cm extends into right pleural space with right pleural effusion [Figure 4]. The patient was treated with antibiotics. Chest drain for pleural effusion and ERCP with sphincterotomy and PD stenting done with a 5Fr × 8 cm single pigtail stent. Pleural effusion resolved and ICD was removed after 7 days. [Table 1] illustrates the summary of the four patients with pancreatico pleural fistula.
Figure 4: Computed tomography film showing right pleural effusion and mediastinal collection communicating into right pleural space

Click here to view
Table 1: Summary of the four patients with pancreatico-pleural fistula

Click here to view


Review of literature

PPF is an abnormal communication between the pancreas and other organs due to leakage of pancreatic secretions from the damaged pancreatic ducts. The incidence is about 0.4% in patients presenting with pancreatitis.[1] In 70%–90% of cases, they occur as a complication of chronic pancreatitis.[2],[3],[4]

In 1976, Cameron et al. coined the term internal pancreatic fistula where the pancreatic secretions drained into a body cavity rather than into the duodenum.[5] Although PPF is rare, it should be considered in the differential diagnosis of patients with pancreatitis who present with recurrent or persistent respiratory symptoms and pleural effusions.[6] A high pleural fluid amylase levels usually indicate pancreatitis as a cause, but it can also be seen in esophageal perforation, parapneumonic effusion, and lung or pancreatic malignancy also.[1],[6],[7],[8] However, only PPF causes very high pleural fluid amylase levels >50,000 IU/L [1],[6]

Patients with PPF may even have recurrent lobar lung consolidation.[6],[9] If the pancreatic ductal rupture occurs anteriorly and is not walled off, then a pancreatico-peritoneal fistula develops resulting in ascites.[10] However, if the ductal disruption occurs posteriorly then the pancreatic collection may dissect through the aortic or esophageal hiatus into the mediastinum to form a pleural fistula or may result in a mediastinal pseudocyst. This mediastinal pseudocyst may further rupture into the pleural cavity leading on to PPF.[10],[11],[12] Imaging studies and pleural fluid analysis usually aids in diagnosing PPF. A PPF may be missed during ERCP if there is a PD stricture or obstruction proximal to the fistula.[6],[13],[14] Performing CT immediately after ERCP may increase the sensitivity of detecting PPF. MRCP has become an important tool in diagnosing PPF as demonstrated in various studies.[15],[16],[17],[18],[19]

Medical treatment may be attempted for about 2–3 weeks before any interventional therapy in places where the expertise is not easily available or when the patient is not very symptomatic or when the demonstrable leak is very small and patient responding to the conservative management.[3]

Somatostatin analogs can significantly reduce fistula output and hasten the closure of both internal and external pancreatic fistulas.[10] Conservative treatment is reported to have an efficacy of 30%–60%.[3]

Endoscopic pancreatic stenting is an effective therapeutic option and in combination with somatostatin analogs such as octreotide, can definitely result in the closure of the fistula and symptomatic improvement.[12] Successful resolution of a PPF after pancreatic stenting was first reported by Saeed et al.[20] Many case reports are available demonstrating the efficacy of the endoscopically placed pancreatic stenting in the treatment of PPF.[21],[22],[23],[24],[25],[26] Pancreatic ductal stenting leads to a pathway of least resistance for the pancreatic secretions to flow into the duodenum, thereby causing the fistulous tract to heal.[20] Applying low intermittent suction through the endoscopic placement of a nasopancreatic drain has also been reported in literature.[27],[28]

Patients with single strictures may be managed successfully with pancreatic endotherapy, whereas patients of multiple strictures, complete duct disruption, or large cysts may be managed with surgery. The most common surgical procedure reported is distal pancreatectomy followed by pancreaticojejunostomy.[29],[30] The best therapeutic approach for the management of PPF patients presenting with MPD obstruction is pancreatic endotherapy along with medical management.


  Conclusion Top


Pancreatic-pleural fistula is a rare complication of pancreatitis and requires a high index of suspicion in patients presenting with chest symptoms or pleural effusion. Extremely high pleural fluid amylase levels are usual but not universally present. Chest X-ray, pleural fluid analysis, and abdominal imaging (MRCP/MRI abdomen more useful than CECT abdomen) would clinch the diagnosis. ERCP with stent/sphincterotomy should be considered when PD reveals a stricture or when medical management fails in patients with dilated or irregular PD. Surgical intervention may be indicated in patients with complete disruption of PD or multiple strictures.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Rockey DC, Cello JP. Pancreaticopleural fistula. Report of 7 patients and review of the literature. Medicine (Baltimore) 1990;69:332-44.  Back to cited text no. 1
    
2.
Broe PJ, Cameron JL. Pancreatic ascites and pancreatic pleural meffusions. In: Bradley EI, editor. Complications of Pancreatitis: Medical and Surgical Management. 1st ed. Philadelphia, PA: WB Saunders; 1982. p. 245.  Back to cited text no. 2
    
3.
Gómez-Cerezo J, Barbado Cano A, Suárez I, Soto A, Ríos JJ, Vázquez JJ, et al. Pancreatic ascites: Study of therapeutic options by analysis of case reports and case series between the years 1975 and 2000. Am J Gastroenterol 2003;98:568-77.  Back to cited text no. 3
    
4.
Kaman L, Behera A, Singh R, Katariya RN. Internal pancreatic fistulas with pancreatic ascites and pancreatic pleural effusions: Recognition and management. ANZ J Surg 2001;71:221-5.  Back to cited text no. 4
    
5.
Cameron JL, Kieffer RS, Anderson WJ, Zuidema GD. Internal pancreatic fistulas: Pancreatic ascites and pleural effusions. Ann Surg 1976;184:587-93.  Back to cited text no. 5
    
6.
François CJ, Demos TC, Iqbal N. Pancreaticothoracic fistulas: Imaging findings in five patients. Abdom Imaging 2005;30:761-7.  Back to cited text no. 6
    
7.
Fulcher AS, Capps GW, Turner MA. Thoracopancreatic fistula: Clinical and imaging findings. J Comput Assist Tomogr 1999;23:181-7.  Back to cited text no. 7
    
8.
Ondrejka P, Faller J, Siket F, Tóth G, Sugár I, Forgács B, et al. Isolated massive pleural effusion caused by pancreatico-pleural fistula. Z Gastroenterol 2000;38:583-5.  Back to cited text no. 8
    
9.
Markos J, Tribe AE, McGonigle P. Recurrent lobar infiltrate and chronic pancreatitis. Med J Aust 1986;145:94-6.  Back to cited text no. 9
    
10.
Ridgeway MG, Stabile BE. Surgical management and treatment of pancreatic fistulas. Surg Clin North Am 1996;76:1159-73.  Back to cited text no. 10
    
11.
Ondrejka P, Siket F, Sugár I, Faller J. Pancreatic-pleural fistula demonstrated by endoscopic retrograde cholangiopancreatography. Endoscopy 1996;28:784.  Back to cited text no. 11
    
12.
Dhebri AR, Ferran N. Nonsurgical management of pancreaticopleural fistula. JOP 2005;6:152-61.  Back to cited text no. 12
    
13.
Nordback I, Sand J. The value of the endoscopic pancreatogram in peritoneal or pleural pancreatic fistula. Int Surg 1996;81:184-6.  Back to cited text no. 13
    
14.
Wakefield S, Tutty B, Britton J. Pancreaticopleural fistula: A rare complication of chronic pancreatitis. Postgrad Med J 1996;72:115-6.  Back to cited text no. 14
    
15.
Tajima Y, Fukuda K, Tsuneoka N, Tsutsumi R, Kuroki T, Onizuka S, et al. Demonstration of a pancreaticopleural fistula with magnetic resonance cholangiopancreatography. Am J Surg 2004;187:741-2.  Back to cited text no. 15
    
16.
Akahane T, Kuriyama S, Matsumoto M, Kikuchi E, Kikukawa M, Yoshiji H, et al. Pancreatic pleural effusion with a pancreaticopleural fistula diagnosed by magnetic resonance cholangiopancreatography and cured by somatostatin analogue treatment. Abdom Imaging 2003;28:92-5.  Back to cited text no. 16
    
17.
Guibaud L, Bret PM, Reinhold C, Atri M, Barkun AN. Bile duct obstruction and choledocholithiasis: Diagnosis with MR cholangiography. Radiology 1995;197:109-15.  Back to cited text no. 17
    
18.
Soto JA, Barish MA, Yucel EK, Siegenberg D, Ferrucci JT, Chuttani R, et al. Magnetic resonance cholangiography: Comparison with endoscopic retrograde cholangiopancreatography. Gastroenterology 1996;110:589-97.  Back to cited text no. 18
    
19.
Taourel P, Bret PM, Reinhold C, Barkun AN, Atri M. Anatomic variants of the biliary tree: Diagnosis with MR cholangiopancreatography. Radiology 1996;199:521-7.  Back to cited text no. 19
    
20.
Saeed ZA, Ramirez FC, Hepps KS. Endoscopic stent placement for internal and external pancreatic fistulas. Gastroenterology 1993;105:1213-7.  Back to cited text no. 20
    
21.
Neher JR, Brady PG, Pinkas H, Ramos M. Pancreaticopleural fistula in chronic pancreatitis: Resolution with endoscopic therapy. Gastrointest Endosc 2000;52:416-8.  Back to cited text no. 21
    
22.
Materne R, Vranckx P, Pauls C, Coche EE, Deprez P, Van Beers BE, et al. Pancreaticopleural fistula: Diagnosis with magnetic resonance pancreatography. Chest 2000;117:912-4.  Back to cited text no. 22
    
23.
Shah HK, Shah SR, Maydeo AP, Pramesh CS. Pancreatico-pleural fistula. Endoscopy 1998;30:314.  Back to cited text no. 23
    
24.
Garcia-Ricart F, Croizet O, El Riwini M, Escourrou J. Endoscopic management of a persistent pancreatopleural fistula. Gastrointest Endosc 1997;46:359-61.  Back to cited text no. 24
    
25.
Iglesias JI, Cobb J, Levey J, Rosiello RA. Recurrent left pleural effusion in a 44-year-old woman with a history of alcohol abuse. Chest 1996;110:547-9.  Back to cited text no. 25
    
26.
Griesshammer B, Strobel M. Pancreaticopleural fistula treated by transpapillary implantation of a plastic prosthesis. Endoscopy 1998;30:741.  Back to cited text no. 26
    
27.
Miyachi A, Kikuyama M, Matsubayashi Y, Kageyama F, Sumiyoshi S, Kobayashi Y, et al. Successful treatment of pancreaticopleural fistula by nasopancreatic drainage and endoscopic removal of pancreatic duct calculi: A case report. Gastrointest Endosc 2004;59:454-7.  Back to cited text no. 27
    
28.
Hastier P, Rouquier P, Buckley M, Simler JM, Dumas R, Delmont JP, et al. Endoscopic treatment of wirsungo-cysto-pleural fistula. Eur J Gastroenterol Hepatol 1998;10:527-9.  Back to cited text no. 28
    
29.
Yanagië H, Tani T, Sairennji T, Ogata M, Eriguchi M. A pancreatic pseudocyst with pancreatic pleural effusion: Report of a case. Surg Today 1997;27:1064-8.  Back to cited text no. 29
    
30.
Chan KL, Lau WY, Sung JY. Octreotide therapy for pancreaticopleural fistula. J Gastroenterol Hepatol 1994;9:530-2.  Back to cited text no. 30
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]
 
 
    Tables

  [Table 1]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Reports
Conclusion
References
Article Figures
Article Tables

 Article Access Statistics
    Viewed672    
    Printed4    
    Emailed0    
    PDF Downloaded111    
    Comments [Add]    

Recommend this journal